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描绘17q24.2 - q24.3微缺失综合征的表型。

Delineating the 17q24.2-q24.3 microdeletion syndrome phenotype.

作者信息

Lestner Jodi M, Ellis Richard, Canham Natalie

机构信息

North West Thames Regional Genetics Unit, London, UK.

出版信息

Eur J Med Genet. 2012 Dec;55(12):700-4. doi: 10.1016/j.ejmg.2012.08.003. Epub 2012 Aug 24.

Abstract

We present an 11-year-old girl with a 2.3 Mb de novo interstitial deletion in chromosome 17q24.2-q24.3 identified by array CGH. The phenotype in this case includes skeletal malformations (lower limb bowing, progressive scoliosis and dental abnormalities), feeding problems, mild learning difficulties, and a characteristic facial appearance. Much of the phenotype is attributable to the deletion of KCNJ2, which causes Andersen Tawil Syndrome (ATS), but the facial appearance is not typical. We hypothesise that the presence of mild channelopathy-related features seen in ATS may be explained by haplo-insufficiency, leading to a reduced number of functionally normal Kir2.1 channels. Comparison is made to previous reports describing overlapping 17q deletions, and potential candidate genes which account for the specific phenotypic similarities with this case are highlighted.

摘要

我们报告了一名11岁女孩,通过阵列比较基因组杂交(array CGH)鉴定出其17号染色体q24.2-q24.3区域存在2.3 Mb的新发间质性缺失。该病例的表型包括骨骼畸形(下肢弯曲、进行性脊柱侧弯和牙齿异常)、喂养问题、轻度学习困难以及特征性面容。该表型的大部分归因于KCNJ2基因的缺失,其可导致安德森-陶威尔综合征(ATS),但面容并不典型。我们推测,在ATS中所见的轻度通道病相关特征可能由单倍体不足所致,导致功能正常的Kir2.1通道数量减少。我们将此病例与之前描述17q重叠缺失的报告进行了比较,并突出了与该病例具有特定表型相似性的潜在候选基因。

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